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142


Severe Agnathia-Otocephaly Complex: Surgical Management and Longitudinal Follow-Up of 4 Patients from Birth Through Adulthood

Alperovich, Michael; Golinko, Michael S; Shetye, Pradip; Flores, Roberto L; Staffenberg, David A
ORIGINAL:0013175
ISSN: 1529-4242
CID: 3589922

Treacher Collins Syndrome and Tracheostomy: Decannulation Utilizing Mandibular Distraction Osteogenesis

Nardini, Gil; Staffenberg, David; Seo, Lauren; Shetye, Pradip; McCarthy, Joseph G; Flores, Roberto L
ORIGINAL:0013185
ISSN: 1529-4242
CID: 3590032

Basal View Reference Photographs for Nasolabial Appearance Rating in Unilateral Cleft Lip and Palate

Rubin, Marcie S; Lowe, Kristen M; Clouston, Sean; Shetye, Pradip R; Warren, Stephen M; Grayson, Barry H
The Asher-McDade system is a 5-point ordinal scale frequently used to rate the components of nasolabial appearance, including nasal form and nasal symmetry, in unilateral cleft lip and palate. Although reference photographs illustrating this scale have been identified for the frontal and right profile view, no reference photographs exist for the basal view. The aim of this study was to identify reference photographs for nasal form and nasal symmetry from the basal view to illustrate this scale and facilitate its use. Four raters assessed nasolabial appearance (form and symmetry) on basal view photographs of 50 children (average age 8 years) with a repaired cleft lip. Intraclass correlation coefficients show fair to moderate inter-rater reliability. Cronbach alpha indicated strong agreement between raters (0.77 nasal form; 0.78 nasal symmetry; 0.80 overall), along with low duplicate measurement error and strong internal consistency between the measures. The photographs with the highest agreement among raters were selected to illustrate each point on the 5-point scale for nasal form and for nasal symmetry, resulting in the selection of 10 reference photographs. The basal view reference photograph set developed from this study may complement existing reference photograph sets for other views and facilitate rating tasks.
PMID: 26163840
ISSN: 1536-3732
CID: 1668602

A comparative study of 3D nasal shape in unilateral cleft lip and palate noses following rotation-advancement and nam-cutting primary nasal repair [Meeting Abstract]

Hosseinian, B; Almaidhan, A; Shetye, P; Cutting, C; Grayson, B
Background & Purpose: The aim of this study was to compare 3D symmetry of the nose in patients with UCLP, subsequent to rotation advancement (Millard) without primary nasal repair and the NAM/Cutting primary nasal repair. Methods & Description: Nasal casts were made for 12 consecutively appearing patients with UCLP, in each of two groups. Group 1 patients had a Millard repair without primary nasal repair (Bardach) while Group 2 patients had NAM and primary nasal repair. Patients were 6 to 18 years of age (mean=12.04). Surgery was performed at the mean age of 3.8 months. None of patients in Group 1 had primary nasal surgery as it was believed at the time by the surgeon that nasal growth might be inhibited. A two flap palatoplasty was performed at 12-24 months (mean age 19.75). All operations were performed by one surgeon in Group 1 and another surgeon in Group 2. Nasal casts were scanned using the 3Shape e scanner. All noses were scaled to the same size prior to evaluation. Procrustes analysis of 3D nasal symmetry was performed using 3dMD Vultus software. The Procrustes technique, determines nasal symmetry by performing a superimposition of its surface with its mirror image (ref Maull 1999). 4 linear measurements including columellar height, nasal dome height, alar base and nasal projections were performed on cleft and non-cleft side in both groups (ref Cutting 1984). For 3D analysis, student's t-test was used to determine the difference between the mean asymmetry index for each group. If symmetry is perfect the asymmetry index is zero. For linear analysis, student's T test was utilized to compare the differences. SPSS was used to perform a descriptive analysis of the groups. Results: The mean asymmetry index in the Millard rotation advancement repair was 4.41 and the NAM plus primary nasal repair was 2.45. The difference was statistically significant (P=0.006). In linear measurements, columellar length and alar base were significantly different when cleft side was compared to non-cleft side in Millard group (P=0.04 and 0.005). There was no significant difference in columellar length, nasal dome height, alar base and nasal projection in cleft versus non-cleft side in NAM group. Inter-group analysis showed that alar base in cleft and non-cleft side is significantly different in Millard versus NAM group (P=0.02). Conclusions: To our knowledge this is the first long-term, quantitative 3D study to analyze the asymmetry of the nose in the Millard rotation advancement versus NAM plus primary nasal repair in patients with complete UCLP. This study shows that the NAM plus primary nasal repair results in significantly less asymmetry of the nose compared to the Millard rotation advancement without nasal correction
EMBASE:617894732
ISSN: 1545-1569
CID: 2682252

Comparative Study of Early Secondary Nasal Revisions and Costs in Patients With Clefts Treated With and Without Nasoalveolar Molding

Patel, Parit A; Rubin, Marcie S; Clouston, Sean; Lalezaradeh, Frank; Brecht, Lawrence E; Cutting, Court B; Shetye, Pradip R; Warren, Stephen M; Grayson, Barry H
The present study aims to determine the risk of early secondary nasal revisions in patients with complete unilateral and bilateral cleft lip and palate (U/BCLP) treated with and without nasoalveolar molding (NAM) and examine the associated costs of care. A retrospective cohort study from 1990 to 1999 was performed comparing the risk of early secondary nasal revision surgery in patients with a CLP treated with NAM and surgery (cleft lip repair and primary surgical nasal reconstruction) versus surgery alone in a private practice and tertiary level clinic. The NAM treatment group consisted of 172 patients with UCLP and 71 patients with BCLP, whereas the non-NAM-prepared group consisted of 28 patients with UCLP and 5 with BCLP. The risk of secondary nasal revision for patients with UCLP was 3% in the NAM group and 21% in the non-NAM group. The risk of secondary nasal revision for patients with BCLP was 7% in the NAM group compared with 40% in the non-NAM group. Using multicenter averages, the non-NAM revision rates were calculated at 37.8% and 48.5% for U/BCLP, respectively. Applying these risks of revision, NAM treatment led to an estimated savings of between $491 and $4893 depending on the type of cleft. In conclusion, NAM can reduce the number of early secondary nasal revision surgeries and, therefore, reduce the overall cost of care.
PMID: 26080163
ISSN: 1536-3732
CID: 1632252

Unilateral Craniofacial Microsomia: Unrecognized Cause of Pediatric Obstructive Sleep Apnea

Szpalski, Caroline; Vandegrift, Meredith; Patel, Parit A; Appelboom, Geoffrey; Fisher, Mark; Marcus, Jeffrey; McCarthy, Joseph G; Shetye, Pradip R; Warren, Stephen M
Bilateral craniofacial microsomia causes obstructive sleep apnea (OSA). We hypothesize that unilateral craniofacial microsomia (UCFM) is an underappreciated cause of OSA. The records of all pediatric UCFM patients from 1990 to 2010 were reviewed; only complete records were included in the study. UCFM patients with OSA (apnea hypopnea index >1/hr) were compared to UCFM patients without OSA. Univariate and multivariate Fisher and chi tests were performed. Of the 62 UCFM patients, 7 (11.3%) had OSA. All OSA patients had Pruzansky IIB or III mandibles. OSA patients presented with snoring (71.4%), failure to thrive (FTT) (57.1%), and chronic respiratory infections (42.8%). Snoring (P < 0.001), Goldenhar syndrome (P = 0.001), and FTT (P = 0.004) were significantly associated with OSA, but race, obesity, clefts, respiratory anomalies, adenotonsillar hypertrophy, and laterality were not. The prevalence of OSA in UCFM patients is up to 10 times greater than in the general population. Snoring, Goldenhar syndrome, and FTT are significantly associated with the presence of OSA.
PMID: 26080175
ISSN: 1536-3732
CID: 1704042

Assessment of Presurgical Clefts and Predicted Surgical Outcome in Patients Treated With and Without Nasoalveolar Molding

Rubin, Marcie S; Clouston, Sean; Ahmed, Mohammad M; M Lowe, Kristen; Shetye, Pradip R; Broder, Hillary L; Warren, Stephen M; Grayson, Barry H
Obtaining an esthetic and functional primary surgical repair in patients with complete cleft lip and palate (CLP) can be challenging because of tissue deficiencies and alveolar ridge displacement. This study aimed to describe surgeons' assessments of presurgical deformity and predicted surgical outcomes in patients with complete unilateral and bilateral CLP (UCLP and BCLP, respectively) treated with and without nasoalveolar molding (NAM). Cleft surgeon members of the American Cleft Palate-Craniofacial Association completed online surveys to evaluate 20 presurgical photograph sets (frontal and basal views) of patients with UCLP (n = 10) and BCLP (n = 10) for severity of cleft deformity, quality of predicted surgical outcome, and likelihood of early surgical revision. Five patients in each group (UCLP and BCLP) received NAM, and 5 patients did not receive NAM. Surgeons were masked to patient group. Twenty-four percent (176/731) of surgeons with valid e-mail addresses responded to the survey. For patients with UCLP, surgeons reported that, for NAM-prepared patients, 53.3% had minimum severity clefts, 58.9% were anticipated to be among their best surgical outcomes, and 82.9% were unlikely to need revision surgery. For patients with BCLP, these percentages were 29.8%, 38.6%, and 59.9%, respectively. Comparing NAM-prepared with non-NAM-prepared patients showed statistically significant differences (P < 0.001), favoring NAM-prepared patients. This study suggests that cleft surgeons assess NAM-prepared patients as more likely to have less severe clefts, to be among the best of their surgical outcomes, and to be less likely to need revision surgery when compared with patients not prepared with NAM.
PMCID:4289121
PMID: 25534051
ISSN: 1049-2275
CID: 1415912

Soft-Tissue Profile Changes following Early Le Fort III Distraction in Growing Children with Syndromic Craniosynostosis

Shetye, Pradip R; Caterson, Edward J; Grayson, Barry H; McCarthy, Joseph G
BACKGROUND: The purpose of this study was to characterize soft-tissue profile changes following Le Fort III (midface) distraction in growing patients with syndromic craniosynostosis. METHODS: The cohort consisted of 20 syndromic patients who underwent Le Fort III osteotomy with midface advancement using a rigid external distraction device. The mean age at surgery was 5.7 years (range, 3 to 12.5 years). Lateral cephalograms were obtained preoperatively (time 1), after distraction device removal (time 2), and 1 year after distraction (time 3). Ten skeletal hard-tissue and 11 soft-tissue profile landmarks were identified and digitized at time points 1, 2, and 3. The x and y displacement of each landmark was studied to determine the ratios for soft- to hard-tissue change. RESULTS: The horizontal ratio of soft- to hard-tissue change for nasal dorsum to orbitale was 0.73:1 and the soft-tissue tip of nose to the anterior nasal spine was 0.86:1. The horizontal ratio of soft-tissue A point to hard-tissue A point was 0.88:1. The horizontal ratio of the upper lip position to the labial surface of maxillary incisor was 0.88:1. The ratio for nasal tip elevation to the anterior nasal spine advancement was 0.27:1. CONCLUSIONS: The result of this study supported the hypothesis that there exists a linear relationship between soft- and hard-tissue changes in the horizontal direction for the midface landmarks following Le Fort III distraction. However, there was a nonlinear relationship between soft- and hard-tissue changes in the vertical direction. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.
PMID: 24076685
ISSN: 1529-4242
CID: 556182

Surgical Management of Patients with a History of Early Le Fort III Advancement after They Have Attained Skeletal Maturity

Caterson, E J; Shetye, Pradip R; Grayson, Barry H; McCarthy, Joseph G
BACKGROUND: The classic Le Fort III procedure was recommended in syndromic craniosynostotic children to reduce exorbitism, improve airway function, and decrease dysmorphism. This study reports on a cohort of syndromic craniosynostosis patients who have undergone early primary subcranial (classic Tessier) Le Fort III advancement and who have been followed longitudinally through skeletal maturity and beyond. METHODS: In this study, the Le Fort III advancements all occurred between the ages of 3 to 5 years, with a mean age of 4.6 years. Subsequently, these early Le Fort III patients were followed throughout development with longitudinal dental, medical, radiographic, and photographic evaluations conducted through skeletal maturity and beyond. For study inclusion, the patients had to have preoperative medical photographs and cephalometric studies at 6 months and 1, 5, and 10 years postoperatively after the primary Le Fort III advancement as well as cephalometric documentation 6 months and 1 year after the secondary midface advancement after skeletal maturity. RESULTS: After early or primary Le Fort III advancement, there was no evidence of relapse and only minimal anterior or horizontal postoperative growth of the midface. However, there was also a return of occlusal disharmony from "anticipated" mandibular growth, approaching a maximum at skeletal maturity. The dysmorphic concave facial profile and malocclusion, and airway and ocular considerations, provided the impetus for secondary midface surgery after skeletal maturity was attained. CONCLUSION: The data demonstrate that early Le Fort III advancement performed before the age of mixed dentition does not obviate the need for a secondary advancement after skeletal maturity is reached. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.
PMID: 24076707
ISSN: 1529-4242
CID: 556202

Orthodontist's Role in Orthognathic Surgery

Wirthlin, John O; Shetye, Pradip R
Orthognathic surgery can eliminate severe esthetic and functional deformities and be a life-changing event for a patient. An orthodontist's role in orthognathic surgery can be divided into several phases: the initial evaluation, presurgical orthodontics, surgical planning, and postsurgical orthodontics. At each of these phases, collaboration between the orthodontist and the surgeon is critical. The ability of an orthodontist and a surgeon to coordinate their efforts during this time is what will lead to a successful outcome.
PMCID:3805727
PMID: 24872759
ISSN: 1535-2188
CID: 1019042